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 Table of Contents  
ORIGINAL ARTICLE
Year : 2022  |  Volume : 9  |  Issue : 1  |  Page : 30-35

Oro-facial clefts at a tertiary level hospital of eastern Nepal: A retrospective insight


1 Department of Oral and Maxillofacial Surgery, B. P. Koirala Institute of Health Sciences, Dharan, Nepal
2 Department of Oral and Maxillofacial Surgery, Kathmandu Medical College, Kathmandu, Kirtipur, Nepal
3 Department of Plastic Surgery, Nepal Cleft and Burn Center, Kathmandu, Kirtipur, Nepal
4 Department of Food and Nutrition, Central Campus of Technology, Dharan, Nepal
5 Department of Oral and Maxillofacial Surgery, Ninewells Hospital, Dundee, Scotland, UK

Date of Submission04-Aug-2021
Date of Acceptance07-Oct-2021
Date of Web Publication01-Jan-2022

Correspondence Address:
Dr. Mehul R Jaisani
Department of Oral and Maxillofacial Surgery, B. P. Koirala Institute of Health Sciences, Dharan
Nepal
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jclpca.jclpca_29_21

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  Abstract 


Introduction: There are only a few centers providing care to orofacial cleft patients across Nepal. However, no adequate data have been reported regarding the epidemiology, pattern, and association of cleft in children from eastern Nepal. The aim of the study was to study the epidemiological profile of cleft lip palate (CLP) patients reporting to the department of oral and maxillofacial surgery (OMFS) in Dharan, Eastern Nepal. Materials and Methods: Data retrospectively retrieved from the cleft registry in the department of OMFS were tabulated and entered into Microsoft Excel 2000. Data of all the diagnosed nonsyndromic orofacial cleft patients attending BP Koirala Institute of Health Sciences for treatment from August 2011 to August 2017 were used for the analysis. Incompletely filled records and syndromic cases were excluded. Results: Data of 220 orofacial clefts were analyzed which included 119 males and 101 females. Among these children with cleft, only 7 patients had direct family members and 18 patients had distant family members having the same condition. Among patients with CL (n = 151), left-sided CL (n = 85) was a more common presentation. Amongst patients with a cleft of the hard plate (n = 153), bilateral (n = 87) presentation was more common. A similar pattern was observed for cleft alveolus (n = 117) with the left side (n = 60) being common. In clefts affecting the soft palate (n = 192), complete cleft (n = 189) was a more common occurrence than incomplete. The majority of our patients had a CLP (n = 133) with isolated CL being the least commonly seen (n = 18). Conclusion: The majority of our patients had a CLP with isolated CL being the least common. This observation is in concordance with reports from other Caucasian and Asian populations both from developed and developing countries.

Keywords: Alveolus, cleft, lip, palate


How to cite this article:
Jaisani MR, Dongol A, Acharya P, Yadav AK, Sagtani A, Pradhan L, Jaisani PV, Laverick S. Oro-facial clefts at a tertiary level hospital of eastern Nepal: A retrospective insight. J Cleft Lip Palate Craniofac Anomal 2022;9:30-5

How to cite this URL:
Jaisani MR, Dongol A, Acharya P, Yadav AK, Sagtani A, Pradhan L, Jaisani PV, Laverick S. Oro-facial clefts at a tertiary level hospital of eastern Nepal: A retrospective insight. J Cleft Lip Palate Craniofac Anomal [serial online] 2022 [cited 2022 Jan 25];9:30-5. Available from: https://www.jclpca.org/text.asp?2022/9/1/30/333644




  Introduction Top


Cleft lip-palate (CLP) is one of the most common congenital malformations. CLP represent a multifactorial malformation including genetic disorders and multiple nutritional and toxicological factors. CLP is a gap which occurs when the lip or roof of the mouth does not fuse completely during the first trimester of fetal development. The lip and palate develop separately and a child may develop cleft lip (CL) or cleft palate (CP) or both. The cleft may be single sided (unilateral) or both sided (bilateral). CP may be restricted to the soft palate or may extend to the hard palate and lip. A family history of CLP has an increased risk for the development of CLP; whilst in CP, the risk is the same compared to the general population.[1]

Few studies have reported on the epidemiological profile of patients with CLP from Nepal, however, the data from the eastern region are sparse. Such a hospital-based investigation can indirectly reflect their access to the health-care facility. The objective of the study was to analyze the epidemiological profile of CLP patients reporting to a tertiary care hospital.


  Materials and Methods Top


Study population

All the diagnosed nonsyndromic cases of orofacial clefts attending/treated at BPKIHS, Eastern Nepal by the Department of Oral and maxillofacial surgery (OMFS).

Exclusion criteria

Incomplete records in the cleft registry and syndromic orofacial clefts.

Study area

B. P. Koirala Institute of Health Sciences, Dharan.

Instrument/tool

The pro forma designed by Smile Train to maintain the cleft registry was used to obtain the epidemiological data.

Ethical

The study was approved by IRC BPKIHS (163/074/075/-IRC Code: IRC/1180/017) and all the patients consented for their data to be used for scientific research. The study was done in accordance with the Helsinki Declaration.

Procedure and analysis

A hospital-based descriptive study was conducted in which 220 CL ± P patients registered under the Smile Train Project from august 2011 to August 2017 in the Department of OMFS, BPKIHS. This study included all the CL ± P patients registered during these 6 years. Informed consent was already taken from the patients/their parents at the time of the data registry. Ethics permission was granted. All the data were collected through a structured questionnaire and entered into Microsoft Excel 2000 version and frequencies were analyzed.


  Results Top


The data were evaluated from 220 patients with orofacial cleft which included 119 males (n = 119; 54.%) and 101 females (n = 101;46%). Mothers of 199 patients did not report having any complications during pregnancy, however, 6 were unsure. Similarly, 213 questionnaires reported no complications during birth and 5 were unsure. When questioned about deleterious habits such as smoking and alcohol consumption during pregnancy, the majority reported not having any (95.9%{211} and 90.9% {200}, respectively). Alcohol consumption was found to be more common than smoking (16 and 5, respectively). Only 3% (7) patients were related to a sibling or parent with CLP. Similarly, only 18 patients had relatives having CLP. When evaluating CL [Table 1] and [Table 2], a unilateral cleft was more common (n = 118) than bilateral (n = 33). In patients having unilateral CL, the left side was observed to be more prevalent (n = 85) over the right side (n = 33), [Figure 1]. On the contrary, bilateral CP (n = 87) was more common than non-cleft and unilateral CP (67, 66), respectively, [Figure 2]. When evaluating clefts of the soft palate, 189 patients had complete clefts, whereas 28 patients had no cleft of the soft palate, [Figure 3].
Table 1: Pattern of unilateral cleft lip and cleft palate (n=220)

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Table 2: Pattern of bilateral cleft lip and cleft alveolus

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Figure 1: Pattern of cleft lip

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Figure 2: Pattern of cleft palate

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Figure 3: Pattern of cleft in soft palate

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[Figure 4] shows the observations for cleft alveolus, where 86 patients had unilateral cleft alveolus and 31 had bilateral [Table 2]. Evaluating the different clefting presentations, we observed CLP to be more prevalent (133) than an isolated cleft of the palate and lip (69, 18 respectively), [Figure 5]. Clinical presentation of few have the cases have shown in [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11].
Figure 4: Pattern of cleft alveolus

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Figure 5: Pattern of cleft lip and cleft palate

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Figure 6: B/L cleft lip, alveolus and palate with severely protruding premaxilla

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Figure 7: Left complete cleft lip, alveolus and palate

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Figure 8: Isolated cleft palate with submucous cleft of hard palate

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Figure 9: Left incomplete cleft lip

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Figure 10: Isolated cleft palate

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Figure 11: Isolate cleft palate with synechia

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  Discussion Top


Although a previous study[2] from BPKIHS reported a higher frequency of females to males with clefts and isolated CL as the most common occurrence. We instead noted a higher frequency of boys with nonsyndromic orofacial clefts compared to girls. This observation is in accordance with observations made in different studies.[3],[4],[5]

Saxén[6] reported a significant association between oro-facial clefts and threatened abortion during the second trimester, concluding that threatened abortion might be a symptom of an already malformed embryo, rather than a cause of the clefting. In this study, the majority of the mothers did not report having any complications during their pregnancy, in contrary to a report from Uganda[3] where 20% of mothers reported threatened abortion. Similarly, the majority of mothers did not report having any complications during birth. However, other studies[3],[7] report having preterm births and infants of low birth weight.

Multiple family members with cleft is very common among CL/P patients, and their relatives have a high risk compared to the general population, but the risk decreases with increasing genetic distance between relatives.[8] It is suggested that oro-facial clefts are inherited conditions implicating the role of heredity.[9],[10] In a study reported from the Philippines,[11] only 3% of CLP patients reported having parents/siblings with cleft. Recurrence rates in siblings for nonsyndromic clefts of the lip and palate were 23/1000 for CL with or without CP, and 14/1000 for CP. Only 8% of the cleft patient mothers reported having relatives with CLP which was in contrary to other reported studies.[3],[10],[12] A 28% positive family history of cleft have been previously reported from Nepal.[13] Wanjeri and Wachira also found that 3.5% of patients had an associated family history of this malformation.[14] Calzolari et al.[15] stated that the complex model of inheritance and the frequently conflicting results in different populations on the role of genes that constitute risk factors suggest the presence of real biologic differences. The complex interplay between genetic and environmental factors undoubtedly plays a role in the pathogenesis of CLP. Hence, relative proportions may vary across different studies.[16]

The etiology of CL/P is attributed to genetic susceptibility and to maternal exposure to environmental risk factors, including smoking, alcohol consumption, medications, and vitamin deficiencies during pregnancy.[17] Our population had a very low association with alcohol and smoking as the majority of the mothers denied any such habits. The frequency of smoking was only 2.27% which was much less than studies reported from Brazil (11.6%),[18] Sweden (23%),[19] and Scotland and England (45%).[20] Similarly, alcohol consumption was reported only in 7.27% of mothers in contrary to the report from Brazil (22.5%).[18] Contrastingly, in a study reported by Donkor et al. from Kumsi, none of the mothers reported smoking cigarettes or consuming alcohol during pregnancy.[21]

The types of clefts can greatly vary between different geographical regions. CL associated with CP was our most common presentation, as also reported from Iran.[22] However, in Brazil,[23] and Bolivia,[24] the most common cleft was CL. Similarly, CP was the most common in Northern Finland, which is in accordance with other studies from Scandinavia.[25]

The unilateral presentation was more common in CL over bilateral with left sided a common occurrence as also noted in world literature.[16],[21]

This study has some inherent limitations, also documented in similar studies.[16] Because of the retrospective study design, missing data on certain cases could not be retrieved. Generalizing the study design may not be possible to the entire population due to the study design and limited sample size. Collection of data through interviews after the birth of patients is likely to be associated with decreasing reliability and with inaccurate answers due to forgetfulness or unrealistic response as a result of being overcome with shyness.[18] Similarly, as mothers were not available to interview in a few cases, data thus obtained may not be fully reliable. Nutritional assessment was beyond the objective of the study.


  Conclusion Top


The majority of our patients had CLP with isolated CL being the least, which is a similar observation reported in studies from Caucasian and Asian populations. Our observations are similar to the reports from developed countries. This study could help in understanding the pattern and demographics of the cleft in this part of the world and raise awareness in the community for access to cleft care. Furthermore, it would provide the foundation for future researches from the center.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Khan M, Ullah H, Naz S, Ullah T, Khan H, Tahir M, et al. Patterns of Cleft Lip and Palate in Norther Pakistan.Arch Clin Exp Surg. 2012; 1:63-70.  Back to cited text no. 1
    
2.
Singh VP, Sharma JN, Roy DK, Roy RK. A study of orofacial clefts seen in a tertiary referral hospital in Nepal. Ceylon Med J 2012;57:84-5.  Back to cited text no. 2
    
3.
Kesande T, Muwazi LM, Bataringaya A, Rwenyonyi CM. Prevalence, pattern and perceptions of cleft lip and cleft palate among children born in two hospitals in Kisoro district, Uganda. BMC Oral Health 2014;14:104.  Back to cited text no. 3
    
4.
Costello BJ, Ruiz RL. Cleft lip and palate: Comprehensive treatment planning and primary repair. In: Miloro M, Ghali GE, Larsen PE, Waite PD, editors. Peterson's Textbook of Maxillofacial Surgery. Hamilton, London: BC Decker Inc.; 2004. p. 841-3.  Back to cited text no. 4
    
5.
Annette M, Travis TT. Cleft deformities in Zimbabwe, Africa: Socioeconomic factors, epidemiology, and surgical reconstruction. Arch Facial Plastic Surg 2007;9:385-91.  Back to cited text no. 5
    
6.
Saxén I. Epidemiology of cleft lip and palate. An attempt to rule out chance correlations. Br J Prev Soc Med 1975;29:103-10.  Back to cited text no. 6
    
7.
Pantaloni M, Bryd HS. Cleft lip 1, primary deformities. Sel Readings Plast Surg 2001;9:5-7.  Back to cited text no. 7
    
8.
Grosen D, Chevrier C, Skytthe A, Bille C, Mølsted K, Sivertsen A, et al. A cohort study of recurrence patterns among more than 54,000 relatives of oral cleft cases in Denmark: Support for the multifactorial threshold model of inheritance. J Med Genet 2010;47:162-8.  Back to cited text no. 8
    
9.
Spritz RA, Arnold TD, Buonocore S, Carter D, Fingerlin T, Odero WW, et al. Distribution of orofacial clefts and frequent occurrence of an unusual cleft variant in the Rift Valley of Kenya. Cleft Palate Craniofac J 2007;44:374-7.  Back to cited text no. 9
    
10.
Fathallah ZF. Effects of socioeconomic factors on the incidence and pattern of orofacial clefts. Basrah J Surg 2007;3:13-21.  Back to cited text no. 10
    
11.
Murray JC, Daack-Hirsch S, Buetwo KH, Munger R, Espina L, Paglinawan N, et al. Clinical and epidemiologic studies of cleft lip and palate in the Philipines. Cleft Palate Craniofac J 1997;34:7-10.  Back to cited text no. 11
    
12.
Owens JR, Jones JW, Harris F. Epidemiology of facial clefting. Arch Dis Child 1985;60:521-4.  Back to cited text no. 12
    
13.
Khatun S. Study of cleft lip and palate in Kathmandu model hospital, Nepal. Orthod J Nepal 2019;9:6-10.  Back to cited text no. 13
    
14.
Wanjeri JK, Wachira JM. Cleft lip and palate: A descriptive comparative, retrospective, and prospective study of patients with cleft deformities managed at 2 hospitals in Kenya. J Craniofac Surg 2009;20:1352-5.  Back to cited text no. 14
    
15.
Calzolari E, Bianchi F, Rubini M, Ritvanen A, Neville AJ; EUROCAT Working Group. Epidemiology of cleft palate in Europe: Implications for genetic research. Cleft Palate Craniofac J 2004;41:244-9.  Back to cited text no. 15
    
16.
Prabakaran S, Thilagam KK, Reddy GM. Profile of cleft lip and cleft palate at a public hospital in southern India. Indian Pediatr 2019;56:753-5.  Back to cited text no. 16
    
17.
Dixon MJ, Marazita ML, Beaty TH, Murray JC. Cleft lip and palate: Understanding genetic and environmental influences. Nat Rev Genet 2011;12:167-78.  Back to cited text no. 17
    
18.
Silva HP, Arruda TT, Souza KS, Bezerra JF, Leite GC, Brito ME, et al. Risk factors and comorbidities in Brazilian patients with orofacial clefts. Braz Oral Res 2018;32:e24.  Back to cited text no. 18
    
19.
Nilsson S, Merlo J, Lyberg-Åhlander V, Psouni E. Psychotropic drug use in adolescents born with an orofacial cleft: A population-based study. BMJ Open 2015;5:e005306.  Back to cited text no. 19
    
20.
Little J, Cardy A, Arslan MT, Gilmour M, Mossey PA. Smoking and orofacial clefts: A United Kingdom-based case-control study. Cleft Palate Craniofac J 2004;41:381-6.  Back to cited text no. 20
    
21.
Donkor P, Plange-Rhule G, Amponsah EK. A prospective survey of patients with cleft lip and palate in Kumasi. West Afr J Med 2007;26:14-6.  Back to cited text no. 21
    
22.
Kianifar H, Hasanzadeh N, Jahanbin A, Ezzati A, Kianifar H. Cleft lip and palate: A 30-year epidemiologic study in north-east of Iran. Iran J Otorhinolaryngol 2015;27:35-41.  Back to cited text no. 22
    
23.
Martelli-Júnior H, Bonan PR, Santos RC, Barbosa DR, Swerts MS, Coletta RD. An epidemiologic study of lip and palate clefts from a Brazilian reference hospital. Quintessence Int 2008;39:749-52.  Back to cited text no. 23
    
24.
McLeod NM, Urioste ML, Saeed NR. Birth prevalence of cleft lip and palate in Sucre, Bolivia. Cleft Palate Craniofac J 2004;41:195-8.  Back to cited text no. 24
    
25.
Lithovius RH, Ylikontiola LP, Harila V, Sándor GK. A descriptive epidemiology study of cleft lip and palate in Northern Finland. Acta Odontol Scand 2014;72:372-5.  Back to cited text no. 25
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]
 
 
    Tables

  [Table 1], [Table 2]



 

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